Abstract

Purpose

Project aims include the following: (i) to identify reported utility values associated with CP in children aged ≤ 18 years; (ii) to explore utility value elicitation techniques in published studies; and (iii) to examine performance of the measures and/or elicitation approaches.

Methods

Peer-reviewed studies published prior to March 2017 were identified from six electronic databases. Construct validity, convergent validity, responsiveness, and reliability of instruments were assessed.

Results

Five studies met the inclusion criteria. Utility values of hypothetical general CP states obtained from a general population of parents ranged from 0.55 to 0.88 using time trade off (TTO) and 0.60–0.87 using standard gamble (SG) techniques. Utility values reported by clinicians of three hypothetical spastic quadriplegic CP states, using the Health Utility Index Mark 2 (HUI-2), ranged from 0.40 to 0.13. Other sources of utilities identified were based on both proxy and child ratings using Health Utility Index Mark 3 (HUI-3) (values ranged from − 0.013 to 0.84 depending on the valuation source) and the Assessment of Quality of Life 4 Dimension instrument, with values ranging from 0.01 to 0.58. Construct validity of the HUI-3 varied from moderate to strong, whereas mixed results were found for convergent validity. Responsiveness and reliability were not reported.

Conclusion

There was substantial variation in reported utilities. Indirect techniques (i.e. via multi-attribute utility instruments) were more frequently used than direct techniques (e.g. TTO, SG). Further research is required to improve the robustness of utility valuation of health-related quality of life in children with CP for use in economic evaluation.

Keywords

Utility value Children Adolescent Cerebral palsy Quality of life Quality-adjusted life years Utility weight

Notes

Funding

This study was funded by the Centre of Research Excellence in Cerebral Palsy (NHMRC APP 1057997) for supporting the conduct of this systematic review. The author UT has received PhD scholarship from the Centre of Research Excellence in Cerebral Palsy.

Compliance with ethical standards

Conflict of interest

The authors declare that they have no conflict of interest.

Human and animal participants

This article does not contain any studies with human participants performed by any of the authors.

Papaioannou, D., Brazier, J., & Parry, G. (2011). How valid and responsive are generic health status measures, such as the EQ-5D and SF-36, in schizophrenia? A systematic review. Value Health: The Journal of the International Society for Pharmacoeconomics and Outcomes Research, 14, 907–920.CrossRefGoogle Scholar

Hawthorne, G., Richardson, J., & Osborne, R. (1999). The Assessment of Quality of Life (AQoL) instrument: A psychometric measure of health-related quality of life. Quality of Life Research, 8(3), 209–224.CrossRefGoogle Scholar

Varni, J. W., Limbers, C. A., & Burwinkle, T. M. (2007). How young can children reliably and validly self-report their health-related quality of life? An analysis of 8,591 children across age subgroups with the PedsQL™ 4.0 Generic Core Scales. Health and Quality of Life Outcomes, 5(1), 1.CrossRefGoogle Scholar

53.

Eiser, C. (1995). Choices in measuring quality of life in children with cancer: A comment. Psycho-Oncology, 4(2), 121–131.CrossRefGoogle Scholar

Canadian Agency for Drugs and Technologies in Health. (2006). Guidelines for the economic evaluation of health technologies (3rd ed.). Ottawa: Canadian Agency for Drugs and Technologies in Health.Google Scholar